Suppr超能文献

与红细胞增多症相关的遗传性丙酮酸激酶异常。

Hereditary pyruvate kinase abnormalities associated with erythrocytosis.

作者信息

Rosa R, Max-Audit I, Izrael V, Beuzard Y, Thillet J, Rosa J

出版信息

Am J Hematol. 1981;10(1):47-55. doi: 10.1002/ajh.2830100108.

Abstract

A familial erythrocytosis is described. The propositus is a 52-year-old man who was found, on routine testing, to have an erythrocytosis with a Hb level of 19.8 g/dl. Physical examination and laboratory findings revealed an associated hemolysis. The P50 of the stripped hemolysis was normal. The 2,3-DPG level was reduced to 15% of normal but that of ATP was increased more than twofold. Red cell diphosphoglycerate mutase activity was normal as were the levels of all red cell glycolytic and hexose monophosphate shunt enzymes with the exception of pyruvate kinase (PK). The level of the latter was elevated to 24.5 U/g (normal: 6.2 +/- 1.1). The electrophoretic pattern of hemolysate PK was abnormal, showing three additional bands, one of these migrated as the M2 isozyme. The findings were similar in the propositus's daughter and her daughter and in one of the two brothers of the propositus. The second brother and the son of the first brother was exhibited erythrocytosis but their red cell 2,3-DPG levels were normal. In addition, the level of their RBC PK was reduced to between 50% and 60% of normal and the abnormal electrophoretic bands were absent. The erythrocytosis appears to be inherited as an autosomal-dominant trait. The relationship between the PK abnormalities appears complex.

摘要

描述了一种家族性红细胞增多症。先证者是一名52岁男性,在常规检查中发现患有红细胞增多症,血红蛋白水平为19.8g/dl。体格检查和实验室检查结果显示伴有溶血。脱纤维蛋白溶血的P50正常。2,3 -二磷酸甘油酸(2,3-DPG)水平降至正常的15%,但三磷酸腺苷(ATP)水平增加了两倍多。红细胞二磷酸甘油酸变位酶活性正常,除丙酮酸激酶(PK)外,所有红细胞糖酵解和磷酸戊糖途径酶的水平也正常。后者的水平升高至24.5U/g(正常:6.2±1.1)。溶血产物PK的电泳图谱异常,显示出另外三条带,其中一条迁移为M2同工酶。先证者的女儿及其女儿以及先证者的两个兄弟中的一个也有类似发现。第二个兄弟和第一个兄弟的儿子表现出红细胞增多症,但他们的红细胞2,3-DPG水平正常。此外,他们的红细胞PK水平降至正常的50%至60%,且没有异常电泳带。红细胞增多症似乎作为常染色体显性性状遗传。PK异常之间的关系似乎很复杂。

文献AI研究员

20分钟写一篇综述,助力文献阅读效率提升50倍。

立即体验

用中文搜PubMed

大模型驱动的PubMed中文搜索引擎

马上搜索

文档翻译

学术文献翻译模型,支持多种主流文档格式。

立即体验